tissue expansion for apert`s syndactyly

TISSUE EXPANSION
FOR APERT'S SYNDACTYLY
D. ASHMEAD and P. J. SMITH
From the Hospitalfor Sick Children, Great Ormond Street, London, UK
Tissue expansion is useful in post-traumatic reconstruction in the upper extremity. Its use has also
been proposed in congenital syndactyly. Expanded local skin flaps would in theory provide locally
appropriate cover, obviating the need for skin grafts. We report a retrospective assessment of
tissue expansion in the management of Apert's syndactyly.:Despite theoretical benefits, tissue
expansion significantly increased the required number of operations. The technique was associated
with an unacceptable rate of complications, and generated inadequate skin flaps, and web spaces
requiring a higher rate of revision than traditional techniques. Despite expectations, tissue expansion for Apert's syndactyly proved disappointing and is not advocated.
Journal of Hand Surgery (British and European Volume, 1995) 20B: 3:327-330
used in combination with traditional flap.and grafting
procedures for the management of Apert's syndactyly.
Individual web spaces were separated by either of the
two techniques, frequently employing a combination
within the same hand. Details of operative technique
varied, but were consistent in principle. Webs treated by
local flap and grafting procedures were separated in a
single stage in the traditional manner. Interdigitating
triangular flaps were arranged for maximum cover and
skin grafts were applied to residual open areas. Primarily
full thickness (groin crease) and rarely split thickness
(thigh or buttock) grafts were employed. Webs treated
by tissue expansion underwent staged reconstruction.
Tissue expanders were introduced subcutaneously with
remote expansion ports, using both internal and externaltypes. After suitable delay for soft tissue healing, expansion was initiated on an out-patient basis, being performed once or twice a week, at Great Ormond Street
when possible. Parents or general practitioners were
instructed in expansion techniques when distances were
great. Expansion was continued until skin flaps appeared
adequate for separation, or when there were complications. The child was then readmitted to hospital for
digital separation, and removal of the expander or its
replacement if necessary. In an effort to expedite digital
separation and to minimize the number of general
anesthetics, separation of one web space was frequently
combined with introduction of an expander elsewhere
in the hand. In the case of traditional flap and grafting
procedures, multiple web spaces were frequently divided
concurrently.
Analysis of the results is based on medical record
review and examination of 20 Apert's children. At the
time of follow-up, all children were still under the care
of the hand service for management of undivided webs,
as well as interval reassessment and revision of previously created web spaces.
Data were summarized in terms of the number of web
spaces created by each technique, as well as the need
for subsequent procedures to maintain web space depth
(revision). Surgery was recorded by the number of
anaesthetics, (not including dressing changes), operations (surgical approaches to a given hand), and
In the reconstruction of congenital syndactyly, local soft
tissues are rarely adequate to allow direct closure of the
separated digits. Although skin grafts will fill the
resulting skin defects, they are not a perfect solution to
the problem. Infection, haematoma, and motion may
interfere with graft take, and scar contraction and graft
pigmentation may compromise functional and aesthetic
outcomes. Some defects, for example where there is
bone or joint exposure, are not amenable to simple
grafting. Preliminary skeletal separation with silicone
sheeting (Stefansson and Stilwell, t 994) may allow grafting of these wounds at final separation. Distant skin
flaps such as the groin flap (Zuker et al, 1991) or the
reverse radial forearm island flap (Fereshetian and
Upton, 1991) may be used, but expansion of adjacent
skin presents an appealing alternative. Reported expansion techniques have included the web space 'pincer'
(Ogawa et al, 1989), a transosseous distraction apparatus (Gudushauri and Tvaliashvili, 1991), and subcutaneous balloons (Borenstein et al, 1991; Morgan
and Edgerton, 1985; Coombs, 1994). The first two
techniques, designed specifically for syndactyly, remain
experimental. The last technique has been successfully
applied in other parts of the upper limb (Wieslander,
1991; Van Beek and Adson, 1987; Carneiro and
Dichiara, 1991; Mackinnon and Gruss, 1985; Meland
et al, 1992) though there are few reports of its use in
syndactyly.
The complex syndactyly of Apert's syndrome appears
to provide an appropriate application for tissue expansion. Involvement of multiple adjacent web spaces
increases the skin re@irement; complex skeletal involvement may lead to bone, cartilage or joint exposure at
the time of separation. Expanded local skin flaps would,
in theory, provide locally appropriate cover while obviating the need for grafts. The use of this technique for
Apert's syndactyly has not been widely discussed; we
have undertaken a retrospective review of the cases
treated at Great Ormond Street.
MATERIALS AND METHODS
Between 1985 and 1989 at the Hospital for Sick
Children, Great Ormond Street, tissue expansion was
327
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328
THE JOURNAL OF HAND SURGERY VOL. 20B No. 3 JUNE 1995
procedures (surgical approaches to a given web space).
This breakdown was necessary in order to account for
simultaneous approaches to both hands or multiple
simultaneous web spaces.
RESULTS
At time of follow-up, 114 web spaces had been treated
by tissue expansion or traditional techniques. The anatomical distribution is summarized in Table 1. All four
web spaces are represented in both categories of surgical
approach, although tissue expansion was used less frequently than traditional techniques for first and fourth
web space separations.
Of 70 web spaces treated by local flap and grafting
techniques, all 70 were successfully divided; nine by
local flaps alone and 61 with the use of skin grafts. 44
spaces were treated with tissue expansion. Of these, 17
(39%) were released employing the expanded skin flaps.
24 web spaces (55%) were found at surgery to have
inadequately expanded flaps; 17 (39%) required grafting
and the balance were closed incompletely or 'under
tension'. In three webs (7%), the expander was removed
without proceeding to web space separation (Table 2).
The 70 web spaces released by traditional techniques
required 57 general anaesthetics and a total of 83
procedures, including revisions. In contrast, the 41 web
spaces released by tissue expansion required 73 general
anaesthetics and a total of 118 procedures including
revisions (Table 3). 67 of these procedures (57%) consisted of expander insertion, adjustment, or removal
alone, without adjunctive web space creation.
Complications are summarized in Table 4. Flap and
Table 3
Tissue expansion
Local flaps and grafts
41 webspaces released
73 anaesthetics
102 operations
118 procedures (2.9/webs)
70
57
69
83
webspaces released
anaesthetics
operations
procedures ( 1.2/web)
Table 4--Complications
Tissue expansion
Leakage
Exposure
Infection
Hematoma
Revision
14
4
3
1
12
Local flaps and grafts
(32%)
(9%)
(7%)
(2%)
(27%)
Graft loss
3
(required re-operation)
Revision
10
(7%)
(14%)
grafting procedures required re-operation for partial
graft loss in three cases (7% of webs) and delayed
revision in ten webs (14%). Complications of tissue
expansion included leakage, exposure, infection and
haematoma, with an aggregate rate of 50%. Revision
was subsequently required in 12 web spaces (27%).
Web space revision rates are broken down more
specifically in Table 5. Flap and grafting procedures led
to a revision rate of 10%, tissue expansion supplemented
by skin grafts 29%, tissue expanded flaps alone 35%,
and unexpanded local flaps 45%. In fact these were part
of a series of planned procedures. Statistical significance
was noted only between the first and fourth categories.
DISCUSSION
Table 1
Web space treated
With tissue
expansion
Without tissue
expansion
Thumb - Index
Index - Middle
Middle Ring
Ring - Little
3
17
11
13
16
20
10
24
Total
44
70
Table 2
Tissue expansion
17 webs released (39%)
24 webs inadequate expansion
Local flaps and grafts
61 released with grafts and flaps
9 released with flaps alone
(55%)
17 grafted
7 partial closure only
3 webs left unseparated (7%)
44 webs spaces treated
70 web spaces treated
Traditional flap and grafting procedures led to predictable separation of 70 web spaces with a modest complication rate. Accounting for revision, these web spaces
were created and maintained with a total of 83 procedures, or 1.2 procedures per web space.
Tissue expansion techniques were less predictable. Of
44 web spaces treated, less than 40% were successfully
released with the expanded flaps. In over 50%, expansion
was felt to be inadequate. The technique was associated
with substantial rates of complication and subsequent
need for revision, as a result of which only 41 web
spaces were created and maintained by 118 procedures
(2.9 procedures per web space). Although the nature of
expansion techniques necessitates a doubling of anaesTable 5--Web space revision
Skin grafts
Tissue expansion - Skin grafts
Tissue expansion alone
Local flaps
(unexpanded)
* P<0.02
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6/61
5/17
6/17
4/9
( 10%)*
(29%)
(35%)
(45%)*
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TISSUE EXPANSION FOR APERT'S SYNDACTYLY
thetics and operative procedures (insertion at a first
stage, removal and separation at the second stage), our
experience involved nearly three times as many procedures. This might be acceptable if the resulting web
spaces were of substantially better quality, but revision
rates in this series were twice as high for tissue expansion
(27%) than for traditional techniques (14%), although
statistical significance was not achieved (Fig 1).
Detailed analysis of revision rates suggests that
increased reliance on local tissue is associated with an
increased revision requirement; skin grafts were associated with the lowest revision rates. Although flaps
should provide more durable and more predictable cover
than skin grafts, lack of adequate local tissues, whether
pre-expanded or not, compromises the depth of web
which can be created. A decision to rely on local tissues
alone limits surgical options, while introduction of grafts
affords additional freedom in web space configuration.
A number of factors may contribute to the unimpressive record of tissue expansion for Apert's syndactyly.
Expanders in the hand are subjected to motion and
minor trauma, particularly in patients of this young age
group. The resulting high rate of exposure and leakage
should not be surprising (Fig 2). Strict immobilization
and protective dressings might help, though the
hyperhidrosis and acne (in adolescence) associated with
Apert's leads to increased dressing change requirements.
These conditions also increase the risk of wound infection. The use of external ports in this series may have
contributed to the incidence of infection, though no
such correlation was found. External ports simplify
expansion by avoiding painful and occasionally misdirected needle placement. External ports have been
used without undue complication in other series
Fig 2
Fig 1
Side-by-side comparison in the same patient of second web
spaces created with tissue expansion (right hand) and traditional flap and grafting procedures (left hand). Ring-little
web spaces were created by traditional techniques bilaterally.
Reliance on expanded flaps alone and avoidance of skin grafts
has limited the resulting depth of the right index-middle
web space.
(a) Partially inflated expander in
become exposed over the distal
(b) Bilateral expanders; on the
adequate while on the left, flaps
threatening exposure.
the left hand which has
part of the ring finger.
right side, skin cover is
have become attenuated,
(Meland et al, 1992) in older age groups without associated hyperhidrosis and acne.
Finally, distances travelled by many patients complicated serial expansion and staged reconstruction.
Frequently, the responsibility for expansion and monitoring fell to general practitioners, health visitors, and
family members with little or no prior experience.
In theory, tissue expansion techniques would seem
ideally suited to the complex syndactyly of Apert's
syndrome. The technique should afford abundant skin
flaps, diminishing the requirement for skin grafts and
producing stable web spaces. In practice, tissue expansion significantly increased the required number of operations and anaesthetics, with an increase in cost. The
technique was associated with an unacceptable rate of
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THE JOURNAL OF HAND SURGERY VOL. 20B No. 3 JUNE 1995
330
complications, and in the end generated inadequate skin
flaps, and web spaces requiring a higher rate of revision
than traditional techniques. Tissue expansion is not
recommended for Apert's syndactyly.
In the words of one patient's mother, 'It's a nice idea,
but it doesn't work, does it?'.
References
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Accepted: 20 September 1994
Duffield Ashmead, MD, 85 Seymour Street, #816 Hartford, CT 06106, USA.
© 1995 The British Societyfor Surgery of the Hand
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